RIOX2

Chr 3

ribosomal oxygenase 2

Also known as: JMJD10, MDIG, MINA, MINA53, NO52, ROX

RIOX2 encodes an oxygenase that demethylates histone H3K9me3 to increase ribosomal RNA expression and hydroxylates ribosomal protein L27a, playing a key role in ribosome biogenesis and cell growth. Mutations cause autosomal recessive intellectual developmental disorder with short stature and variable brain malformations. The gene shows very high constraint against loss-of-function variants, indicating that complete loss of function is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 1.07
Clinical SummaryRIOX2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
10 unique Pathogenic / Likely Pathogenic· 97 VUS of 124 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.07LOEUF
pLI 0.000
Z-score 1.31
OE 0.70 (0.461.07)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.05Z-score
OE missense 0.99 (0.891.10)
253 obs / 255.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.70 (0.461.07)
00.351.4
Missense OE0.99 (0.891.10)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 15 / 21.6Missense obs/exp: 253 / 255.5Syn Z: -0.18

ClinVar Variant Classifications

124 submitted variants in ClinVar

Classification Summary

Pathogenic7
Likely Pathogenic3
VUS97
Likely Benign4
7
Pathogenic
3
Likely Pathogenic
97
VUS
4
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
7
0
7
Likely Pathogenic
0
0
3
0
3
VUS
0
86
11
0
97
Likely Benign
0
4
0
0
4
Benign
0
0
0
0
0
Total090210111

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

RIOX2 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗